1 - Department of Neuroradiology, Hospital Dona Estefânia, Centro Hospitalar Universitário Lisboa Central, Lisbon, Portugal.
2 - Pediatrics Infectious Diseases Unit, Hospital Dona Estefânia, Centro Hospitalar Universitário Lisboa Central, Lisbon, Portugal.
- 46th ESNR Annual Meeting, 20 a 24 setembro 2023, Viena
- Neuroradiology (2023) 65 (Suppl 1):S1–S146
- https://doi.org/10.1007/s00234-023-03203-z
Introduction: Mycetoma is a chronic, slowly progressive, destructive granulomatous infection primarily affecting the skin, subcutaneous tissues, and underlying bone. It can be caused by fungi (eumycetoma) or filamentous bacteria (actinomycetoma) and predominantly affects the limbs, back, and gluteal region, due to traumatic inoculation. Young individuals are most affected, with a male predominance explained by occupational exposure in tropical and subtropical regions. Diagnosis is based on clinical presentation and isolation of the causative microorganism. Defining the etiology is important to guide therapy with antibacterials or antifungals, which can be challenging. Imaging findings include granulomatous soft tissue masses and bone changes associated with the chronic nature of the infection. In this article, we present the case of an adolescent with extracranial, intracranial, and orbital mycetoma and discuss its evolution.
Case Report: A 15-year-old boy, with a history of craniofacial trauma 3 years prior, presented with painful inflammatory ulcerated scalp and left superior eyelid lesions,accompanied by purulent discharge. The initial CT scan revealed thickening of the scalp and extensive sclerotic changes in the frontal calvaria, sphenoid bone, and maxilla with transosseous channels. MRI confirmed the presence of bifrontal pachymeningeal thickening/epidural soft tissue components and left intraorbital involvement, leading to mild proptosis. Biopsy disclosed a soft tissue mycetoma of probable fungal origin, but microbiological and molecular biology studies were negative. The patient underwent empirical treatment with antifungals for 18 months, combined with cutaneous surgical debridement. Marked clinical improvement was observed, with healing of the cutaneous lesions. Follow-up imaging showed resolution of the extracranial soft tissue components and skull bone changes, with the disappearance of transosseous channels. Focal residual dural thickening persisted, as well as epicranial sequelae changes.
Discussion & Conclusion: This case report highlights the rarity of a neglected tropical disease, particularly unknown in non-endemic countries beyond the so-called mycetoma belt. The disease's significant morbidity requires early intervention to prevent further progression and associated complications. However, despite prolonged therapeutic regimens, outcomes are often unsatisfactory. The late presentation of these patients to healthcare services is reflected in its imaging findings evocative of a progressive indolent infection. Involvement of the skull and intracranial compartment is uncommon, and this rare case illustrates the imaging findings and subsequent changes observed following treatment.
Keywords: Mycetoma; Head; Skull; Imaging; CT; MRI